Chronic health conditions after childhood Langerhans cell histiocytosis: Results from the Swiss Childhood Cancer Survivor Study.

Sláma, Tomáš; Mader, Luzius; Žarković, Maša; Malär, Reta; Schifferli, Alexandra; von der Weid, Nicolas X; Kuehni, Claudia E; Schindera, Christina (2024). Chronic health conditions after childhood Langerhans cell histiocytosis: Results from the Swiss Childhood Cancer Survivor Study. (In Press). Journal of Cancer Survivorship Springer 10.1007/s11764-024-01544-z

[img]
Preview
Text
s11764-024-01544-z.pdf - Published Version
Available under License Creative Commons: Attribution (CC-BY).

Download (895kB) | Preview

PURPOSE

Langerhans cell histiocytosis (LCH) is a rare disease characterized by dysregulated proliferation of myeloid marrow progenitors and subsequent organ infiltration. While LCH is associated with a favorable prognosis, some survivors may develop chronic health conditions (CHC) because of the disease. In this study, we aimed to assess the spectrum and prevalence of CHC among LCH survivors compared with siblings and identify factors associated with the development of CHC.

METHODS

The Swiss Childhood Cancer Survivor Study sent questionnaires to all ≥ 5-year LCH survivors registered in the Swiss Childhood Cancer Registry and diagnosed between 1976 and 2015. Siblings also received similar questionnaires. We compared CHC prevalence between LCH survivors and siblings and used logistic regression to identify determinants of CHC.

RESULTS

A total of 123 LCH survivors participated in the study, with a response rate of 69%. Median time since diagnosis was 13 years (interquartile range 9-20). Among LCH survivors, 59% had at least one CHC. Cardiovascular (13% vs. 6%), endocrine (15% vs. 2%), musculoskeletal (22% vs. 13%), and digestive (15% vs. 8%) CHC were more common among LCH survivors compared to siblings (all p < 0.05). Factors most strongly associated with the occurrence of CHC were multisystem LCH, multifocal bone involvement, and involvement of the pituitary gland.

CONCLUSIONS

More than half of long-term LCH survivors suffered from one or more CHC and were affected considerably more than siblings.

IMPLICATIONS FOR CANCER SURVIVORS

LCH survivors in follow-up care should be screened especially for cardiovascular, endocrine, musculoskeletal, and digestive conditions.

Item Type:

Journal Article (Original Article)

Division/Institute:

04 Faculty of Medicine > Department of Gynaecology, Paediatrics and Endocrinology (DFKE) > Clinic of Paediatric Medicine
04 Faculty of Medicine > Pre-clinic Human Medicine > Institute of Social and Preventive Medicine (ISPM)

Graduate School:

Graduate School for Cellular and Biomedical Sciences (GCB)
Graduate School for Health Sciences (GHS)

UniBE Contributor:

Sláma, Tomáš, Mader, Luzius Adrian, Zarkovic, Masa, Kühni, Claudia, Schindera, Christina

Subjects:

600 Technology > 610 Medicine & health
300 Social sciences, sociology & anthropology > 360 Social problems & social services

ISSN:

1932-2267

Publisher:

Springer

Language:

English

Submitter:

Pubmed Import

Date Deposited:

15 Feb 2024 10:53

Last Modified:

27 Feb 2024 14:46

Publisher DOI:

10.1007/s11764-024-01544-z

PubMed ID:

38353855

Additional Information:

Claudia E. Kuehni and Christina Schindera contributed equally to this work (shared last authorship).

Open access funding provided by University of Bern.

Uncontrolled Keywords:

Childhood cancer survivors Chronic health conditions Cohort study Langerhans cell histiocytosis Late effects Rare disease

BORIS DOI:

10.48350/192913

URI:

https://boris.unibe.ch/id/eprint/192913

Actions (login required)

Edit item Edit item
Provide Feedback